Purpose: Epileptic negative myoclonus (ENM) has been increasingly reco
gnized in different epilepsies, but the reasons for its appearance and
prognosis remain uncertain. We report 3 patients who developed de nov
o, almost continuous ENM, triggered by antiepileptic drug (AED) taperi
ng, that resolved with treatment. Methods: Three patients aged 16, 19,
and 65 years with a 13- to 36-year history of partial epilepsy were r
eceiving a therapeutic dosage of carbamazepine or phenobarbital plus e
ither clobazam (CLB) or valproate (VPA). None had previously had ENM.
Forty-eight to 72 h after CLB or VPA withdrawal, the habitual seizures
recurred. The patient also began to report repetitive postural lapses
of one or more limbs that interfered with eating or writing. At this
time, each patient underwent polygraphy with simultaneous surface elec
tromyography (EMG) of deltoid, biceps, and triceps muscles and of the
wrist extensor and flexor bilaterally. Results. In all patients, EEGs
demonstrated almost continuous epileptiform discharges whose spatial d
istribution was similar to that observed before ENM appearance. Polygr
aphic recordings showed repetitive loss of postural EMG activity in on
e or more limbs, 100-400 ms in duration, which occurred in conjunction
with the spike-waves. One milligram of clonazepam intravenously alway
s terminated ENM status, which has not recurred in the ensuing 9-36 mo
nths. Conclusions: ENM may emerge as a new type of seizure due to tape
ring of AED therapy. This effect is possibly related to the great acti
vation of epileptiform activity with consequent interference with cort
ical activity.